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Original Report |
1
Department of Radiology, University Hospitals of Cleveland, Case Western
Reserve University, 11100 Euclid Ave., Cleveland, OH 44106.
2
Department of Pathology, Baptist Medical Center, 800 Prudential Dr.,
Jacksonville, FL 32207.
3
Mallinckrodt Institute of Radiology, 510 S. Kingshighway Blvd., St. Louis, MO
63110.
4
Department of Pathology, University Hospitals of Cleveland, Case Western
Reserve University, Cleveland, OH 44106.
5
Baptist Medical Center, Baptist Regional Cancer Institute, 800 Prudential Dr.,
Jacksonville, FL 32207.
Received September 25, 2000;
accepted after revision December 5, 2000.
Address correspondence to S. Abramson.
Abstract
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CONCLUSION. Benign metastasizing leiomyoma is an asymptomatic disease characterized by well-defined, numerous, pulmonary lesions without a preponderant distribution.
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The pathologic features of this entity are described throughout the literature. However, there are only scattered case reports describing chest radiographic and CT findings in the lungs. The purpose of our study was to determine the clinical presentation, chest radiographic findings, and CT findings of benign metastasizing leiomyoma.
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Clinical data were gathered through patient chart review. Patient age, clinical presentation, history of malignancy, and time interval from hysterectomy to discovery of nodules were noted.
Chest radiographic techniques varied from institution to institution, but most were performed using standard posteroanterior projection with a high-kilovoltage technique (125 kVp, 5 mAs, 12:1 grid ratio). CT scans were obtained with a variety of machines, including the PQ5000 (Picker International, Cleveland, OH). CT techniques were not uniform because there was more than one hospital involved in the collection of images. IV contrast medium—enhanced studies were performed in all patients. The chest radiographs and CT scans were reviewed retrospectively by one of two chest radiologists.
Chest radiographs were evaluated for lesion size, distribution, and number.
Associated findings such as lymphadenopathy or pleural effusion were also
noted. A lesion was described as a nodule (<3 cm in diameter) or as a mass
(
3 cm in diameter).
Thoracic CT images were reviewed in a similar pattern to that used for the radiographs, with special attention to lesion size, shape, distribution, and number. Border characteristics (i.e., poor or well defined) and the presence of calcification were also analyzed. In addition, mediastinal or hilar lymphadenopathy and pleural and pericardial involvement were noted.
In all patients, the diagnosis was based on lung biopsy results. One patient underwent a diagnostic percutaneous needle biopsy, and six patients required a surgical procedure. Original microscopic specimens were reviewed in five of the seven patients and the report was reviewed in the remaining two. Original uterine pathology slide specimens were available in three patients and uterine pathology reports were reviewed in two other patients. Uterine pathology was not available in one patient and pulmonary nodules were discovered before hysterectomy in another patient. This data collection was performed independently by two pulmonary pathologists.
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Total hysterectomy had been performed in five patients and myomectomy in one patient before the discovery of pulmonary lesions. The time interval from hysterectomy to discovery of nodules ranged from 4 to 12 years (mean, 8 years). Bilateral oophorectomy and left oophorectomy were conducted in one patient each. Pulmonary nodules were discovered in one patient before hysterectomy.
Diagnostic Procedures
Diagnostic transthoracic percutaneous biopsy with an 18-gauge Tru-Cut
needle (Baxter, Deerfield, IL) was conducted in one patient. One patient
underwent a nondiagnostic transthoracic needle aspiration complicated by a
pneumothorax. Diagnostic tissue was obtained via open thoracotomy (n
= 3) or video-assisted thorascopic surgery with wedge resection (n =
3).
Radiologic Findings
Chest radiographs showed multiple pulmonary lesions ranging in size from a
few millimeters to 4 cm in diameter (Figs.
1A,
2A, and
3A). Well-circumscribed lesions
were scattered throughout the lung without a preponderant distribution. No
pleural effusions or lymphadenopathy were seen.
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On CT, lesions ranged in size from 1 to 42 mm in diameter (Fig. 1B). Multiple subcentimeter nodules throughout both lungs were seen in four patients (Fig. 2B). Most lesions were smoothly marginated with well-defined borders (Figs. 3B and 4). In one patient, CT showed lobulated nodules (Fig. 5A). A pericardial effusion was seen in another patient. No nodules contained calcification. There was no evidence of pleural effusions or significant mediastinal lymphadenopathy.
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Abdominal and pelvic CT images were available in two patients. In one patient, the images showed two large heterogeneous masses with a central area of necrosis (Fig. 5B). The second patient was found to have a 5 x 3.2 cm heterogeneously enhancing right lower pelvis soft-tissue mass after myomectomy. These findings were compatible with a leiomyomatous uterus.
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Pathologic Findings
All five available pulmonary specimens (four wedge biopsies and one cutting
needle biopsy) showed similar features (Fig.
6A,6B).
There was a proliferation of cytologically benign smooth muscle without
anaplasia. Mitotic figures were rare. Larger nodules were circumscribed
without encapsulation, but smaller foci had less discrete contours. Some of
the lesions were less cellular and contained moderate amounts of collagen.
Metaplastic, low cuboidal epithelium was present on alveolar septa at the
periphery of the lesions with extension and entrapment of similarly lined
clefts and tubular spaces in the interior of the nodules. Cytoskeletal
immunochemistry performed in one patient confirmed smooth muscle origin with
strong reactivity for desmin and muscle-specific actin, and weaker reactivity
for vimentin. Samples from two patients were stained for estrogen and
progesterone receptors; findings for both were positive.
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Uterine specimens from three patients were examined. The first, from a hysterectomy 3.5 years before identification of the pulmonary lesions, showed multiple benign leiomyomata with a 10-cm submucosal tumor that showed neither anaplasia nor increased cellularity or mitoses. The second patient had a 7.5-cm leiomyoma removed by myomectomy 5.5 years before the appearance of the lung lesions was noted. This was classified as a cellular leiomyoma because it showed increased cellular density without anaplasia or increased mitotic activity. The third patient had adenomyosis, multiple leiomyomata, and leiomyomatosis peritonealis disseminata. The latter lesion was unusual because the peritoneal smooth-muscle proliferation also contained endometrial glands and stroma, a finding only rarely reported in leiomyomatosis peritonealis disseminata [3]. The smooth-muscle component was histologically benign without anaplasia or increased mitotic activity.
A fourth patient had multiple recurrent benign smooth-muscle tumors in the pelvis, measuring up to 8.5 cm in diameter. These tumors were removed 1 month after the patient had been diagnosed with pulmonary benign metastasizing leiomyomata, which was 7 years after hysterectomy for benign leiomyomata. These pelvic lesions were also classified as benign metastasizing leiomyomata at the original facility, and were reported to show immunochemical reactivity for estrogen and progesterone receptors.
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Although there have been reports of patient morbidity and mortality from benign metastasizing leiomyoma, the clinical course is typically indolent, with patient mortality commonly from an unrelated disease process [5]. In our series, no patient had symptoms despite the presence of severe pulmonary involvement in several patients. Little correlation exists between disease extent and respiratory symptoms [6].
Typical radiographic findings include well-circumscribed solitary or multiple pulmonary nodules ranging in size from a few millimeters to several centimeters in diameter scattered among normal interstitium. A miliary pattern is less common, but was described by Lipton et al. [7]. Additionally, there are single case reports of benign metastasizing leiomyoma manifesting as cavitary lung nodules and interstitial lung disease [8]. Typically, nodules are not calcified and do not enhance after IV contrast medium administration. Pulmonary nodules usually show little change and may even spontaneously regress [9]. Endobronchial and pleural sparing is characteristic.
Although most commonly seen in the lungs, other sites of involvement include lymph nodes, peritoneum, and retroperitoneal structures [3, 10]. A medium-sized pericardial effusion in one of our patients was thought to represent pericardial involvement; however, confirmatory histologic sampling was not performed. Three of our patients had unusual pelvic lesions associated with pulmonary benign metastasizing leiomyoma. The first had recurrent leiomyomata. The second had both uterine leiomyomata and leiomyomatosis peritonealis disseminata. This latter condition is generally regarded as a diffuse metaplastic phenomenon within the peritoneal stroma, rather than extension or metastasis of a uterine tumor [3]. To our knowledge, the association of benign metastasizing leiomyoma with leiomyomatosis peritonealis disseminata has not been previously reported. The third patient had recurrence of morphologically benign pelvic smooth muscle tumors around the same time the pulmonary lesions were identified.
Benign metastasizing leiomyoma should not be confused with lymphangioleiomyomatosis; they are distinct entities. Lymphangioleiomyomatosis is characterized by the proliferation of smooth muscle cells from lymphatic walls in the lung and lymph nodes. Young women can present with a spontaneous pneumothorax, chylous pleural effusion, or progressive dyspnea. Characteristic imaging findings include hyperinflation and numerous thin-walled cystic spaces.
The pathogenesis of benign metastasizing leiomyoma has been a subject of some controversy over the years. In the past, these lesions were termed "multiple fibroleiomyomatous hamartomas" when they were thought to originate in situ in the lungs [9]. The presence of entrapped metaplastic epithelial elements contributed to this confusion. Most pathologists now accept these lesions as hematogenous metastases from morphologically benign uterine tumors, although there is a continuum with lesions that show increased mitotic activity [2]. Some authors have proposed classifying the latter cases as low-grade leiomyosarcoma, but in general these cases behave similarly and are easily distinguished from metastatic high-grade leiomyosarcoma [11]. The benign pathologic specimens of uterine leiomyoma confirm the benign nature of this entity. Additionally, coexistence of a histologically benign uterine tumor with distant metastases of the same composition is a key diagnostic feature [12]. The argument that more extensive sampling of uterine tissue may reveal sarcomatous degeneration does not account for the benign histology of metastatic foci [6].
Despite the high frequency of uterine leiomyomas in the general population, pulmonary metastases are rare. Several mechanisms of spread have been theorized on the basis of a spectrum of pathologic processes. It has been proposed that benign-appearing smooth muscle spreads to the lungs after uterine extension into pelvic venous channels [12]. Alternatively, smooth muscle neoplasms may arise directly from vessel walls. However, the latter is associated with extension into the inferior vena cava and does not result in metastatic foci [6]. Others claim that tumors gain venous access from surgical trauma during hysterectomy [2, 4]. This would not explain visualization of nodules before surgery, a phenomenon seen in one of our patients.
Estrogen and progesterone receptors have been identified in lung lesions and have led to treatment options based on hormonal manipulation through surgical or medical oophorectomy [1, 4]. Lung lesions tend to remain stable with occasional regression after treatment. However, therapy may not always be indicated. The effects of natural hormonal changes such as pregnancy and menopause have also been associated with lesion regression [9].
In summary, benign metastasizing leiomyoma is a rare entity that usually affects women after hysterectomy for leiomyomas. Lesions are incidentally discovered and have an indolent clinical course with patient mortality from an unrelated disease process. The lungs are the most common site of metastatic involvement. On chest radiography and CT, multiple nodules and pulmonary masses can be seen. Mediastinal and hilar lymphadenopathy is rare. Benign metastasizing leiomyoma should be considered in any asymptomatic patient presenting with multiple pulmonary nodules and a history of a leiomyomatous uterus.
Acknowledgments
We thank Virginia M. Wormald for preparation of this manuscript.
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